INTRODUCTION: Vulvovaginal cancer in pregnancy is rare. Limited data complicate decision-making and patient counseling. Our review, coupled with new case data, fills a current gap in the literature and provides practical insights. MATERIAL AND METHODS: Oncological and obstetric data of these pregnancies were examined by a case collection from the International Network on Cancer, Infertility and Pregnancy (INCIP) registry (vulvar n = 10, vaginal n = 5) and a literature review (vulvar n = 46, vaginal n = 37). RESULTS: Although preoperative imaging of inguinofemoral lymph nodes is feasible, only 16.1% of vulvar cancer patients underwent ultrasound or MRI. Treatment was initiated during pregnancy for 69.1% of vulvar cancer and 28.4% of vaginal cancer patients. Surgical lymph node staging of vulvar cancer was postponed until after delivery in 10 cases, although uni- or bilateral lymphadenectomy during pregnancy was not associated with more complications. Delivery outcomes included a live birth rate of 96.4% for vulvar cancer and 50% for vaginal cancer due to the high rate of pregnancy terminations, with most births preterm. The overall 5-year survival rates for vulvar (81.3%) and vaginal (66.4%) cancer during pregnancy are comparable to nonpregnant populations, indicating that pregnancy does not adversely impact maternal prognosis. CONCLUSIONS: This study underscores the feasibility of adapting standard oncological care for pregnant patients, emphasizing multidisciplinary teams to optimize maternal and fetal outcomes.

Amsterdam Reproduction and Development Research Institute Amsterdam The Netherlands

Department of Development and Regeneration KU Leuven Leuven Belgium

Department of Gynaecologic Oncology University of Amsterdam Amsterdam The Netherlands

Department of Gynecologic Oncology Netherlands Cancer Institute Amsterdam The Netherlands

Department of Gynecological Oncology Centre of Gynecological Oncology Amsterdam The Netherlands

Department of Medicine and Surgery University of Milan Bicocca Milan Italy

Department of Obstetrics and Gynecology Amsterdam University Medical Centre Vrije Universiteit Amsterdam The Netherlands

Department of Obstetrics and Gynecology Cooper University Health Care Camden New Jersey USA

Department of Obstetrics Copenhagen University Hospital Rigshospitalet Denmark

Division of Gynecological Oncology Department of Obstetrics and Gynecology UZ Leuven Leuven Belgium

Division of Obstetrics Department of Obstetrics and Gynecology University Hospitals Leuven Leuven Belgium

Medical Oncology Radboud University Medical Centre Nijmegen The Netherlands

National Medical Research Centre for Obstetrics Gynecology and Perinatology Named after Academician 5 1 Kulakov of the Ministry of Healthcare of Russian Federation Moscow Russia

Unit of Gynecological Oncology Department of Oncology KU Leuven Leuven Belgium

University Hospital Kralovske Vinohrady and 3rd Medical Faculty Charles University Prague Czech Republic

UO Gynecology Fondazione IRCCS San Gerardo Dei Tintori Monza Italy

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Dalmartello M, Negri E, La Vecchia C, et al. Frequency of pregnancy‐associated cancer: a systematic review of population‐based studies. Cancers. 2020;12(6):1356. PubMed PMC

Lee Y, Roberts C, Dobbins T, et al. Incidence and outcomes of pregnancy‐associated cancer in Australia, 1994–2008: a population‐based linkage study. BJOG. 2012;119(13):1572‐1582. PubMed PMC

Eibye S, Kjær SK, Mellemkjær L. Incidence of pregnancy‐associated cancer in Denmark, 1977–2006. Obstet Gynecol. 2013;122(3):608‐617. PubMed

Cottreau CM, Dashevsky I, Andrade SE, et al. Pregnancy‐associated cancer: a U.S. population‐based study. J Womens Health (Larchmt). 2019;28(2):250‐257. PubMed PMC

Pilleron S, Sarfati D, Janssen‐Heijnen M, et al. Global cancer incidence in older adults, 2012 and 2035: a population‐based study. Int J Cancer. 2019;144(1):49‐58. PubMed

Lenaerts L, Theunis M, Amant F, Vermeesch JR. Non‐invasive prenatal testing: when results suggests maternal cancer. Med Genet. 2023;35(4):285‐295. PubMed PMC

Lundberg FE, Stensheim H, Ullenhag GJ, et al. Risk factors for the increasing incidence of pregnancy‐associated cancer in Sweden – a population‐based study. Acta Obstet Gynecol Scand. 2024;103(4):669‐683. PubMed PMC

Amant F. Gynecologic cancers in pregnancy: guidelines based on a third international consensus meeting. Ann Oncol. 2019;30:1601‐1612. PubMed

Huang J, Chan SC, Fung YC, et al. Global incidence, risk factors and trends of vulvar cancer: a country‐based analysis of cancer registries. Int J Cancer. 2023;153(10):1734‐1745. PubMed

Huang J, Chan SC, Pang WS, et al. Incidence distributions, risk factors and trends of vaginal cancer: a global population‐based study. BJOG. 2024;131(12):1660‐1672. PubMed

Bray F, Laversanne M, Sung H, et al. Global cancer statistics 2022: GLOBOCAN estimates of incidence and mortality worldwide for 36 cancers in 185 countries. CA Cancer J Clin. 2024;74(3):229‐263. PubMed

WHO Classification of Tumours Editorial Board . Female genital tumours. 5th ed. International Agency for Research on Cancer; 2020.

Lai J, Elleray R, Nordin A, et al. Vulval cancer incidence, mortality and survival in England: age‐related trends. BJOG. 2014;121(6):728‐738. PubMed

Joura EA, Lösch A, Haider‐Angeler MG, Breitenecker G, Leodolter S. Trends in vulvar neoplasia: increasing incidence of vulvar intraepithelial neoplasia and squamous cell carcinoma of the vulva in young women. Obstet Gynecol Surv. 2001;56(1):24‐26. PubMed

Kang YJ, Smith M, Barlow E, Coffey K, Hacker N, Canfell K. Vulvar cancer in high‐income countries: increasing burden of disease. Int J Cancer. 2017;141(11):2174‐2186. PubMed

Rasmussen CL, Thomsen LT, Baandrup L, et al. Time trends in prevalence of p16 positivity and combined HPV/p16 positivity in a large cohort of Danish vulvar cancer patients. Int J Cancer. 2023;152(11):2424‐2432. PubMed

Adams TS, Cuello MA. Cancer of the vagina. Int J Gynaecol Obstet. 2018;143(S2):14‐21. PubMed

Hellman K, Silfverswärd C, Nilsson B, Hellström AC, Frankendal B, Pettersson F. Primary carcinoma of the vagina: factors influencing the age at diagnosis. The Radiumhemmet series 1956–96. Int J Gynecol Cancer. 2004;14(3):491‐501. PubMed

Exposure in utero to diethylstilbestrol and related synthetic hormones: association with vaginal and cervical cancers and other abnormalities. JAMA. 1976;236(10):1107‐1109. PubMed

Lecointre L, Gaudineau A, Hild C, Sananes N, Langer B. Carcinome épidermoïde de la vulve et grossesse: des choix difficiles. Gynecol Obstet Fertil. 2015;43(9):625‐627. PubMed

Gaunt E, Pounds R, Yap J. Vulval cancer in pregnancy: two case reports. Case Rep Womens Health. 2021;33:e00374. PubMed PMC

Olayemi O, Aimakhu CO, Omigbodun AO, Ogunbiyi JO, Udoh IJ. Vulval carcinoma in pregnancy. J Obstet Gynaecol. 2002;22(4):441‐442. PubMed

Keskin N, Iyibozkurt AC, Topuz S, Salihoğlu Y, Bengisu E, Berkman S. Invasive squamous carcinoma of the vulva in women aged less than 40 years: report of two cases and a third case diagnosed during pregnancy. Eur J Gynaecol Oncol. 2008;29(4):399‐401. PubMed

Amant F, Han SN, Gziri MM, Vandenbroucke T, Verheecke M, Van Calsteren K. Management of cancer in pregnancy. Best Pract Res Clin Obstet Gynaecol. 2015;29(5):741‐753. PubMed

Wolters V, Heimovaara J, Maggen C, et al. Management of pregnancy in women with cancer. Int J Gynecol Cancer. 2021;31(3):314‐322. PubMed PMC

Oonk MHM, Planchamp F, Baldwin P, et al. European Society of Gynaecological Oncology Guidelines for the management of patients with vulvar cancer—update 2023. Int J Gynecol Cancer. 2023;33(7):1023‐1043. PubMed PMC

Abu‐Rustum NR, Yashar CM, Arend R, et al. Vulvar cancer, version 3.2024, NCCN Clinical Practice Guidelines in Oncology. J Natl Compr Canc Netw. 2024;22(2):117‐135. PubMed

Nikolić O, Sousa FA, Cunha TM, et al. Vulvar cancer staging: guidelines of the European Society of Urogenital Radiology (ESUR). Insights Imaging. 2021;12(1):131. PubMed PMC

Zapardiel I, Iacoponi S, Coronado PJ, et al. Prognostic factors in patients with vulvar cancer: the VULCAN study. Int J Gynecol Cancer. 2020;30(9):1285‐1291. PubMed

Fischerova D, Garganese G, Reina H, et al. Terms, definitions and measurements to describe sonographic features of lymph nodes: consensus opinion from the Vulvar International Tumor Analysis (VITA) group. Ultrasound Obstet Gynecol. 2021;57(6):861‐879. PubMed

te Grootenhuis NC, van der Zee AGJ, van Doorn HC, et al. Sentinel nodes in vulvar cancer: long‐term follow‐up of the GROningen INternational study on sentinel nodes in vulvar cancer (GROINSS‐V) I. Gynecol Oncol. 2016;140(1):8‐14. PubMed

Levenback CF, Ali S, Coleman RL, et al. Lymphatic mapping and sentinel lymph node biopsy in women with squamous cell carcinoma of the vulva: a gynecologic oncology group study. J Clin Oncol. 2012;30(31):3786‐3791. PubMed PMC

Covens A, Vella ET, Kennedy EB, Reade CJ, Jimenez W, Le T. Sentinel lymph node biopsy in vulvar cancer: systematic review, meta‐analysis and guideline recommendations. Gynecol Oncol. 2015;137(2):351‐361. PubMed

Hassanzade M, Attaran M, Treglia G, Yousefi Z, Sadeghi R. Lymphatic mapping and sentinel node biopsy in squamous cell carcinoma of the vulva: systematic review and meta‐analysis of the literature. Gynecol Oncol. 2013;130(1):237‐245. PubMed

Di Donna MC, Quartuccio N, Giallombardo V, et al. Detection of sentinel lymph node in vulvar cancer using 99mTc‐labeled colloid lymphoscintigraphy, blue dye, and indocyanine‐green fluorescence: a meta‐analysis of studies published in 2010–2020. Arch Gynecol Obstet. 2023;307(6):1677‐1686. PubMed

Koual M, Benoit L, Nguyen‐Xuan HT, Bentivegna E, Azaïs H, Bats AS. Diagnostic value of indocyanine green fluorescence guided sentinel lymph node biopsy in vulvar cancer: a systematic review. Gynecol Oncol. 2021;161(2):436‐441. PubMed

Nijman TAJ, Schutter EM, Amant F. Sentinel node procedure in vulvar carcinoma during pregnancy: a case report. Gynecol Oncol Case Rep. 2012;2(2):63‐64. PubMed PMC

Metke F, Bösken E, Pixberg M, et al. EP1176 treatment of early‐stage vulvar carcinoma in a pregnant woman: a case report. Int J Gynecol Cancer. 2019;29:A608.

Winarno AS, Fehm TN, Hampl M. Vulvar cancer during pregnancy and/or breastfeeding: a report of five cases from a single center study at the University Hospital of Düsseldorf. BMC Pregnancy Childbirth. 2022;22(1):207. PubMed PMC

Ogunleye D, Lewin SN, Huettner P, Herzog TJ. Recurrent vulvar carcinoma in pregnancy. Gynecol Oncol. 2004;95(2):400‐401. PubMed

Nout R, Calaminus G, Planchamp F, et al. ESTRO/ESGO/SIOPE guidelines for the management of patients with vaginal cancer. Radiother Oncol. 2023;186:109662. PubMed

Senekjian EK, Hubby M, Bell DA. Clear cell adenocarcinoma (CCA) of the vagina and cervix in association with pregnancy. Gynecol Oncol. 1986;24(2):207‐219. PubMed

Palmer JP, Biback SM. Primary cancer of the vagina. Am J Obstet Gynecol. 1954;67(2):377‐397. PubMed

Del Castillo H, Rubio PA, Farrell EM. Vaginal adenocarcinoma in a gravida with prenatal DES exposure. Int J Gynaecol Obstet. 1978;16(4):271‐273. PubMed

Lv L, Sun Y, Liu H, Lou J, Peng Z. Neoadjuvant chemotherapy followed by radical surgery and reconstruction of the vagina in a patient with stage II primary vaginal squamous carcinoma. J Obstet Gynaecol Res. 2010;36(6):1245‐1248. PubMed

Benedetti Panici P, Bellati F, Plotti F, et al. Neoadjuvant chemotherapy followed by radical surgery in patients affected by vaginal carcinoma. Gynecol Oncol. 2008;111(2):307‐311. PubMed

Diao Y, Jiao J, Song K, et al. Effects of neoadjuvant chemotherapy on patients with primary vaginal squamous cell carcinoma. Mol Clin Oncol. 2017;7(3):395‐398. PubMed PMC

Cancer Research UK . Vulval cancer incidence statistics [Internet]. 2015. https://www.cancerresearchuk.org/health‐professional/cancer‐statistics/statistics‐by‐cancer‐type/vulval‐cancer/incidence

SEER . Cancer of the vulva – Cancer Stat Facts [Internet]. https://seer.cancer.gov/statfacts/html/vulva.html

Mack LC, Hagemeier A, Forner DM. Influence of stage and age on survival of patients with vulvar cancer in Germany: a retrospective study. BMJ Open. 2024;14(8):e077960. PubMed PMC

Olawaiye AB, Cotler J, Cuello MA, et al. FIGO staging for carcinoma of the vulva: 2021 revision. Int J Gynaecol Obstet. 2021;155(1):43‐47. PubMed PMC

American Cancer Society . Survival rates for vaginal cancer [Internet]. https://www.cancer.org/cancer/types/vaginal‐cancer/detection‐diagnosis‐staging/survival‐rates.html

Gonzalez Bosquet J, Magrina JF, Gaffey TA, et al. Long‐term survival and disease recurrence in patients with primary squamous cell carcinoma of the vulva. Gynecol Oncol. 2005;97(3):828‐833. PubMed